Literature DB >> 25113655

Paediatric hereditary angioedema: a survey of UK service provision and patient experience.

N Read1, E Lim, M D Tarzi, P Hildick-Smith, S Burns, K J Fidler.   

Abstract

Hereditary angioedema (HAE) is a rare disease characterized by episodes of potentially life-threatening angioedema. For affected children in the United Kingdom, there are relatively few data regarding disease prevalence, service organization and the humanistic burden of the disease. To improve knowledge in these areas, we surveyed major providers of care for children with HAE. A questionnaire was sent to major paediatric centres to determine patient numbers, symptoms, diagnostic difficulties, management and available services. In addition, all patients at a single centre were given a questionnaire to determine the experiences of children and their families. Sixteen of 28 centres responded, caring for a total of 111 UK children. Seven children had experienced life-threatening crises. One-third of patients were on long-term prophylactic medication, including C1 inhibitor prophylaxis in four children. Eight centres reported patients who were initially misdiagnosed. Broad differences in management were noted, particularly regarding indications for long-term prophylaxis and treatment monitoring. We also noted substantial variation in the organization of services between centres, including the number of consultants contributing to patient care, the availability of specialist nurses, the availability of home therapy training and the provision of patient information. Ten of 12 patient/carer questionnaires were returned, identifying three common themes: the need to access specialist knowledge, the importance of home therapy and concerns around the direct effect of angioedema on their life. To our knowledge, this study represents the first dedicated survey of paediatric HAE services in the United Kingdom and provides useful information to inform the optimization of services.
© 2014 British Society for Immunology.

Entities:  

Keywords:  C1 esterase; angioedema; hereditary; paediatrics

Mesh:

Year:  2014        PMID: 25113655      PMCID: PMC4238875          DOI: 10.1111/cei.12433

Source DB:  PubMed          Journal:  Clin Exp Immunol        ISSN: 0009-9104            Impact factor:   4.330


  26 in total

1.  Evidence-based recommendations for the therapeutic management of angioedema owing to hereditary C1 inhibitor deficiency: consensus report of an International Working Group.

Authors:  M Cicardi; K Bork; T Caballero; T Craig; H H Li; H Longhurst; A Reshef; B Zuraw
Journal:  Allergy       Date:  2011-11-30       Impact factor: 13.146

Review 2.  The pathophysiology of hereditary angioedema.

Authors:  Alvin E Davis
Journal:  Clin Immunol       Date:  2005-01       Impact factor: 3.969

3.  Ecallantide (DX-88) for acute hereditary angioedema attacks: integrated analysis of 2 double-blind, phase 3 studies.

Authors:  Albert L Sheffer; Marilyn Campion; Robyn J Levy; H Henry Li; Patrick T Horn; William E Pullman
Journal:  J Allergy Clin Immunol       Date:  2011-04-09       Impact factor: 10.793

4.  Icatibant, a new bradykinin-receptor antagonist, in hereditary angioedema.

Authors:  Marco Cicardi; Aleena Banerji; Francisco Bracho; Alejandro Malbrán; Bernd Rosenkranz; Marc Riedl; Konrad Bork; William Lumry; Werner Aberer; Henning Bier; Murat Bas; Jens Greve; Thomas K Hoffmann; Henriette Farkas; Avner Reshef; Bruce Ritchie; William Yang; Jürgen Grabbe; Shmuel Kivity; Wolfhart Kreuz; Robyn J Levy; Thomas Luger; Krystyna Obtulowicz; Peter Schmid-Grendelmeier; Christian Bull; Brigita Sitkauskiene; William B Smith; Elias Toubi; Sonja Werner; Suresh Anné; Janne Björkander; Laurence Bouillet; Enrico Cillari; David Hurewitz; Kraig W Jacobson; Constance H Katelaris; Marcus Maurer; Hans Merk; Jonathan A Bernstein; Conleth Feighery; Bernard Floccard; Gerald Gleich; Jacques Hébert; Martin Kaatz; Paul Keith; Charles H Kirkpatrick; David Langton; Ludovic Martin; Christiane Pichler; David Resnick; Duane Wombolt; Diego S Fernández Romero; Andrea Zanichelli; Francesco Arcoleo; Jochen Knolle; Irina Kravec; Liying Dong; Jens Zimmermann; Kimberly Rosen; Wing-Tze Fan
Journal:  N Engl J Med       Date:  2010-08-05       Impact factor: 91.245

5.  Economic costs associated with acute attacks and long-term management of hereditary angioedema.

Authors:  David A Wilson; Konrad Bork; Elizabeth P Shea; Anne M Rentz; Marc B Blaustein; William E Pullman
Journal:  Ann Allergy Asthma Immunol       Date:  2010-04       Impact factor: 6.347

6.  Hereditary angioedema due to C1 inhibitor deficiency: patient registry and approach to the prevalence in Spain.

Authors:  Olga Roche; Alvaro Blanch; Teresa Caballero; Noelia Sastre; Daniel Callejo; Margarita López-Trascasa
Journal:  Ann Allergy Asthma Immunol       Date:  2005-04       Impact factor: 6.347

7.  On demand treatment and home therapy of hereditary angioedema in Germany - the Frankfurt experience.

Authors:  Emel Aygören-Pürsün; Inmaculada Martinez-Saguer; Eva Rusicke; Thomas Klingebiel; Wolfhart Kreuz
Journal:  Allergy Asthma Clin Immunol       Date:  2010-07-28       Impact factor: 3.406

8.  Hereditary angio-oedema in Denmark: a nationwide survey.

Authors:  A Bygum
Journal:  Br J Dermatol       Date:  2009-06-22       Impact factor: 9.302

Review 9.  Home management of haemophilia.

Authors:  J M Teitel; D Barnard; S Israels; D Lillicrap; M-C Poon; J Sek
Journal:  Haemophilia       Date:  2004-03       Impact factor: 4.287

10.  Recurrent abdominal pain due to hereditary angioedema.

Authors:  Deepa Janardhanan; Sajitha Nair; T S Subramanian
Journal:  Indian J Pediatr       Date:  2007-01       Impact factor: 5.319

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  4 in total

1.  Current state of hereditary angioedema management: a patient survey.

Authors:  Aleena Banerji; Paula Busse; Sandra C Christiansen; Henry Li; William Lumry; Mark Davis-Lorton; Jonathan A Bernstein; Michael Frank; Anthony Castaldo; Janet F Long; Bruce L Zuraw; Marc Riedl
Journal:  Allergy Asthma Proc       Date:  2015 May-Jun       Impact factor: 2.587

Review 2.  The Humanistic, Societal, and Pharmaco-economic Burden of Angioedema.

Authors:  Hilary Longhurst; Anette Bygum
Journal:  Clin Rev Allergy Immunol       Date:  2016-10       Impact factor: 8.667

3.  Clinical characteristics and real-life diagnostic approaches in all Danish children with hereditary angioedema.

Authors:  Anne Aabom; Klaus E Andersen; Christina Fagerberg; Niels Fisker; Marianne A Jakobsen; Anette Bygum
Journal:  Orphanet J Rare Dis       Date:  2017-03-16       Impact factor: 4.123

Review 4.  Self-administered C1 esterase inhibitor concentrates for the management of hereditary angioedema: usability and patient acceptance.

Authors:  Huamin Henry Li
Journal:  Patient Prefer Adherence       Date:  2016-09-07       Impact factor: 2.711

  4 in total

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